Literature DB >> 21220631

Incidence, ocular findings, and systemic associations of ocular coloboma: a population-based study.

Kelly M Nakamura1, Nancy N Diehl, Brian G Mohney.   

Abstract

OBJECTIVE: To describe the incidence, ocular findings, and systemic associations of coloboma in a population-based cohort of children.
METHODS: We retrospectively reviewed the medical records of pediatric (aged <19 years) patients diagnosed as having ocular coloboma from January 1, 1968, through December 31, 2007, as residents of Olmsted County, Minnesota.
RESULTS: Thirty-three children were newly diagnosed as having ocular coloboma (annual incidence, 2.4 per 100,000 residents <19 years old; prevalence, 1 in 2077 live births). Median patient age at diagnosis was 3.9 months (range, 2 days to 18.4 years), and 22 patients (67%) had unilateral involvement. Twelve patients (36%) had involvement of the anterior segment only, 13 (39%) of the posterior segment only, and 8 (24%) of both. During median ophthalmologic follow-up of 9.2 years (range, 13 days to 35.9 years), 19 patients (58%) had other ocular disorders, including amblyopia in 11 (33%) and strabismus in 10 (30%). During median medical follow-up of 16.8 years, 22 patients (67%) were diagnosed as having a nonocular disorder, including abnormal development in 12 (36%) and CHARGE (coloboma, heart defects, choanal atresia, retarded growth and development, genital abnormalities, and ear anomalies) syndrome in 4 (12%).
CONCLUSIONS: Ocular coloboma occurred in 1 in 2077 live births. More than half of the patients were diagnosed as having an ocular disorder other than coloboma, including strabismus and amblyopia in approximately one-third. Two-thirds of patients were diagnosed as having a nonocular disorder, including CHARGE syndrome in 1 in 8 patients.

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Year:  2011        PMID: 21220631      PMCID: PMC3126628          DOI: 10.1001/archophthalmol.2010.320

Source DB:  PubMed          Journal:  Arch Ophthalmol        ISSN: 0003-9950


  28 in total

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Journal:  Pediatrics       Date:  1996-06       Impact factor: 7.124

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Journal:  Sci Am       Date:  1981-10       Impact factor: 2.142

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Journal:  Indian J Ophthalmol       Date:  1981-12       Impact factor: 1.848

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Journal:  Ophthalmology       Date:  1996-12       Impact factor: 12.079

7.  Congenital iris coloboma repair using a modified McCannel suture technique.

Authors:  Douglas M Blackmon; Scott R Lambert
Journal:  Am J Ophthalmol       Date:  2003-05       Impact factor: 5.258

Review 8.  Congenital eye anomalies.

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Journal:  Pediatr Clin North Am       Date:  2003-02       Impact factor: 3.278

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Journal:  Clin Dysmorphol       Date:  1993-10       Impact factor: 0.816

10.  National study of microphthalmia, anophthalmia, and coloboma (MAC) in Scotland: investigation of genetic aetiology.

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Journal:  J Med Genet       Date:  2002-01       Impact factor: 6.318

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  26 in total

1.  Clinical and Echographic Features of Retinochoroidal and Optic Nerve Colobomas.

Authors:  Vincent D Venincasa; Yasha S Modi; Hassan A Aziz; Bernadette Ayres; Claus Zehetner; Wei Shi; Timothy G Murray; Harry W Flynn; Audina M Berrocal
Journal:  Invest Ophthalmol Vis Sci       Date:  2015-06       Impact factor: 4.799

2.  Two cases of CHARGE syndrome with multiple congenital anomalies.

Authors:  Joo Hyun Chang; Dong Ho Park; Jae Pil Shin; In Taek Kim
Journal:  Int Ophthalmol       Date:  2013-06-27       Impact factor: 2.031

3.  Systemic diagnostic testing in patients with apparently isolated uveal coloboma.

Authors:  Nancy Huynh; Delphine Blain; Tanya Glaser; E Lauren Doss; Wadih M Zein; David M Lang; Eva H Baker; Suvimol Hill; Carmen C Brewer; Jeffrey B Kopp; Tanya M Bardakjian; Irene H Maumenee; Bronwyn J Bateman; Brian P Brooks
Journal:  Am J Ophthalmol       Date:  2013-09-05       Impact factor: 5.258

Review 4.  Relationship between neural crest cell specification and rare ocular diseases.

Authors:  Monica Akula; Jeong Won Park; Judith A West-Mays
Journal:  J Neurosci Res       Date:  2018-04-16       Impact factor: 4.164

5.  EFTUD2 deficiency in vertebrates: Identification of a novel human mutation and generation of a zebrafish model.

Authors:  Brett Deml; Linda M Reis; Sanaa Muheisen; David Bick; Elena V Semina
Journal:  Birth Defects Res A Clin Mol Teratol       Date:  2015-06-27

6.  De novo apparent loss-of-function mutations in PRR12 in three patients with intellectual disability and iris abnormalities.

Authors:  Magalie S Leduc; Marianne Mcguire; Suneeta Madan-Khetarpal; Damara Ortiz; Susan Hayflick; Kory Keller; Christine M Eng; Yaping Yang; Weimin Bi
Journal:  Hum Genet       Date:  2018-03-19       Impact factor: 4.132

7.  Retinitis pigmentosa with bilateral irido-fundal coloboma.

Authors:  Devi Bharathi Daggula; Hima Bindu Adusumilli; Krishna Chaitanya Penmetsa; Venkata Bindu Nekkanti; Avinash Mahindrakar
Journal:  Indian J Ophthalmol       Date:  2020-10       Impact factor: 1.848

Review 8.  Spotlight on the pediatric eye: a pictorial review of orbital anatomy and congenital orbital pathologies.

Authors:  Roopa Bhat; Mohannad Al-Samarraie; Ayman Nada; Carlos Leiva-Salinas; Matthew Whitehead; Eman Mahdi
Journal:  Neuroradiol J       Date:  2020-08-31

9.  Clinical diagnosis of presumed SOX2 gonadosomatic mosaicism.

Authors:  Malena Daich Varela; Robert B Hufnagel; Bin Guan; Delphine Blain; Julie C Sapp; Andrea L Gropman; Ramakrishna Alur; Jennifer J Johnston; Leslie G Biesecker; Brian P Brooks
Journal:  Ophthalmic Genet       Date:  2021-03-15       Impact factor: 1.274

10.  Joubert Syndrome: Ophthalmological Findings in Correlation with Genotype and Hepatorenal Disease in 99 Patients Prospectively Evaluated at a Single Center.

Authors:  Brian P Brooks; Wadih M Zein; Amy H Thompson; Maryam Mokhtarzadeh; Daniel A Doherty; Melissa Parisi; Ian A Glass; May C Malicdan; Thierry Vilboux; Meghana Vemulapalli; James C Mullikin; William A Gahl; Meral Gunay-Aygun
Journal:  Ophthalmology       Date:  2018-07-25       Impact factor: 12.079

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